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Cancer of the Small Intestine in Patients with Crohn's Disease

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Cancer of the Small Intestine in Patients with Crohn's Disease ANTICANCER RESEARCH 33: 2977-2980 (2013) Cancer of the Small Intestine in Patients with Crohn’s Disease DAIJIRO HIGASHI1,4, KITARO FUTAMI1,4, DAIBO KOJIMA1, RYO FUTATSUKI1, YUKIKO ISHIBASHI1, TAKAFUMI MAEKAWA1, YUTAKA YANO2,4, NORITAKA TAKATSU2,4, FUMIHITO HIRAI2,4, TOSHIYUKI MATSUI2,4 and AKINORI IWASHITA3 Departments of 1Surgery, 2Gastroenterology, 3Pathology and 4Inflammatory Bowel Disease Center, Fukuoka University Chikushi Hospital, Chikushino-shi, Fukuoka, Japan Abstract. Due to an increase in the number of long-term strictures in patients with Crohn’s disease. The development cases of Crohn’s disease, the risk of combined cancer in of small intestine cancer in patients with Crohn’s disease is these patients has been assessed in numerous articles. Most very rare; however, when it does occur, it leads to a poor of these reports have involved patients with cancer of the prognosis. Therefore, the diagnosis and treatment of this large intestine, while cases of cancer of the small intestine condition are associated with problems that must be combined with Crohn’s disease are very rare. We addressed. experienced two cases of cancer of the small intestine combined with Crohn’s disease. In both cases, the patients Case Reports had suffered from Crohn’s disease for over 10 years and a second operation was performed after a long period without Case 1 involved a 36-year-old female. She was diagnosed treatment following the first operation, which had achieved a with Crohn’s disease at 23 years of age. She suffered from favorable outcome. In both cases of combined cancer, the diabetes as a complication, and her family history included patients experienced ileus; however, it was difficult to discern a younger sister who also suffered from Crohn’s disease. At this from ileus due to the presence of Crohn’s disease. 25 years of age, she experienced ileus due to stricture, for Therefore, making a definitive diagnosis of combined cancer which she underwent surgery. Because she was diabetic, was not possible before surgery, and the definitive diagnosis intestinal resection was avoided, while strictureplasty and was obtained based on an intraoperative pathological bypass were performed. The patient demonstrated good diagnosis. It is thought that tumor markers transition in a progress after the surgery and visited the hospital once a manner parallel to the progression of cancer, providing a year. Eleven years after the first operation, she began to clue for cancer diagnosis. In patients with Crohn’s disease, suffer from nausea and abdominal pain. A computed there is a pressing need to establish a method for diagnosing tomography (CT) scan was performed, which led to a cancer of the small intestine at an early stage. diagnosis of hypertrophy of the small intestinal wall (Figure 1A). A small bowel series of X-rays was performed, which Cancer of the small intestine occurring in combination with resulted in a diagnosis of expansion of the oral side and the Crohn’s disease is not as common as cancer of the large strictures that frequently occur in the small intestine inside intestine; however, the relative risk is high. In addition, since the pelvis (Figure 2A). Treatment administered via an ileus cancer of the small intestine developing in patients with tube was performed for two weeks. The patient’s subjective Crohn’s disease occurs in infected regions, it is very difficult symptoms, such as nausea and abdominal pain, improved; to detect the disease at an early stage. This is because X-ray however, no improvement was observed in the expansion of images of small intestine cancer are similar to the images of the small intestine on an X-ray examination. Therefore, a second operation was performed. The findings of laparotomy revealed advanced adhesion between the intestines, as well as the presence of nodules in Douglas’ Correspondence to: Daijiro Higashi, Department of Surgery, pouch of the peritoneum. Since these findings were quite Fukuoka University Chikushi Hospital, 1-1-1 Zokumyoin, different from those observed during regular laparotomy in Chikusino-shi, Fukuoka 818-8502, Japan. Tel: +81 929211011, Fax: +81 929280856, e-mail: [email protected] patients with Crohn’s disease, an intraoperative pathological diagnosis was made by evaluating pieces of the intestinal Key Words: Crohn’s disease, small intestine cancer, surgical resection tissue and nodules. The results showed well-to- operation. moderately differentiated adenocarcinoma. Measurement of 0250-7005/2013 $2.00+.40 2977 ANTICANCER RESEARCH 33: 2977-2980 (2013) Figure 1. A computed tomography (CT) image obtained before surgery in case 1 (A), and case 2 (B). Both images show hypertrophy of the small intestinal wall near the lesion (arrow). Figure 2. Results of the small bowel X-ray series in case 1(A), and case 2(B). Both patients display multiple strictures (arrow). It is difficult to distinguish Crohn’s disease from the cancer. the levels of tumor markers during surgery revealed a CA19-9 level returned to normal for a short period. clearly high carbohydrate antigen 19-9 (CA19-9) level of However, in the sixth month after the operation, a CT scan 962 U/ml (Table I). The final pathological diagnosis revealed a pelvic tumor, thought to be a site of recurrence, indicated the presence of cancerous lesions in two locations: that coincided with an increased in the CA19-9 level (Table in a region that was excluded during the bypass procedure I). Chemotherapy was subsequently administered as and in the stricture on the oral side of the bypass (Figure 3A treatment for recurring large intestine cancer; however, an and B). After the operation, treatment with FOLFOX increase in the number of pelvic tumors could not be (levofolinate, fluouracil, oxaliplatin) and bevacizumab was suppressed, resulting in the patient’s death one year and six commenced. Following the initiation of chemotherapy, the months after the operation. 2978 Higashi et al: Small Intestine Cancer in Crohn’s Disease Table I. Transition of tumor markers. Before After surgery surgery 3 Months 6 Months 9 Months Case 1 CEA 2.1 2.2 4.3 19.3 CA19-9 962 32 66 413 Case 2 CEA 13.2 4.0 10.2 15.7 CA19-9 358 60 49 24 CEA: Carcinoembryonic antigen ; CA19-9: carbohydrate antigen 19-9. The second patient was a 45-year-old male. The disease presented as abdominal pain and melena at 22 years of age; therefore, he had been suffering from the disease for 23 years. At 23 years of age, the patient underwent appendectomy due to appendicitis, and at that time was diagnosed with Crohn’s disease. At 25 years of age, ileocecal resection was performed due to stenosis of the ileum. The patient’s progress after the operation was favorable, and 13 years passed without any treatment. At 38 years of age, the patient developed an aching sensation in the right side of the abdomen. For the next seven years, he received medical therapy, primarily centered on prednisolone. Since ileus had also appeared at that time, the patient was admitted to this hospital. The CT findings were similar those of case 1, which lead to a diagnosis of hypertrophy of the small intestinal wall (Figure 1B). A small bowel series of X-rays revealed an anastomotic site in the region of the previous surgery with multiple areas of stenosis on the oral side (Figure 2B). A fistula was also Figure 3. Resected specimens in case 1. Cancer (arrows) was found in found near the anastomotic region, and the intestinal tract the region excluded during the previous bypass operation (A) and the stricture lesion (B) on the oral side of the bypass. Findings of the on the oral side had expanded. During colonoscopy, we resected specimen in case 2 (C). Cancer (arrow) was found in the lesion were unable to insert the scope because the stenosis on the in the stricture on the oral side of the anastomosis (triangle) created oral side was too severe. For this reason, we determined that during the previous operation. the areas of stenosis were due to recurrence of Crohn’s disease in the anastomotic region, and we performed surgery. The area around the anastomotic region was very hard, and nodules were found in the peritoneum. It was Discussion determined that the patient’s symptoms were caused by advanced cancer; therefore, an intraoperative pathological The incidence of Crohn’s disease has steadily increased, with diagnosis was made. The results indicated the presence of both the overall number of patients and the number of long- well-differentiated adenocarcinoma. We performed right term cases increasing. It is known that cancer can develop in hemicolectomy including the area of stenosis. The final the digestive tract, including the small and large intestines, pathological diagnosis was a cancerous lesion at the as a result of long-term chronic inflammation (1-4). The stricture on the oral side of the anastomosis formed during cumulative risk is 0.2% in the first 10 years and 2.2% in the the previous operation (Figure 3C). After surgery, we first 25 years after the onset of Crohn’s disease (5). administered chemotherapy with FOLFOX and The first case of cancer of the small intestine combined bevacizumab. To date, there has been no obvious recurrence with Crohn’s disease was reported by Ginzburg et al., (6) in on imaging examinations. 1956. This condition is not as common as large intestine 2979 ANTICANCER RESEARCH 33: 2977-2980 (2013) cancer combined with Crohn’s disease; however, the relative References risk is very high (7, 8). Cancer of the small intestine combined with Crohn’s disease does not involve the specific 1 Bernstein CN, Blanchard JF, Kliewer E and Wajda A: Cancer symptoms observed at the onset of small intestine cancer and risk in patients with inflammatory bowel disease: A population- does not display spindle-shaped benign strictures without based study.
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